The treatment and prognosis of chronic myeloid leukemia have dramatically changed since the introduction of tyrosine kinase inhibitors, but although several clinical trials have examined their safety with respect to heart function, no data are yet available about the use of these drugs in patients with Brugada syndrome. We report a case of Brugada syndrome diagnosed during tyrosine kinase inhibitor therapy in a 69-year-old Caucasian male with meningioma and chronic myeloid leukemia. This case report highlights the importance of an integrated approach among hematologists and cardiologists to ensure appropriate treatment with tyrosine kinase inhibitors in patients affected by chronic myeloid leukemia who also suffer from Brugada syndrome.